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Tumor-induced Osteomalacia: A Sherlock Holmes Approach to Diagnosis and Management

Tumor-induced osteomalacia (TIO) is a subtype of paraneoplastic syndrome associated with hypophosphatemia due to renal phosphate wasting in adults. The humoral factor responsible for clinical picture known as fibroblast growth factor 23 (FGF23) is most often secreted by benign yet elusive mesenchyma...

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Autores principales: Chanukya, G. V., Mengade, Manoj, Goud, Jagadishwar, Rao, I. Satish, Jain, Anuj
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Medknow Publications & Media Pvt Ltd 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5502504/
https://www.ncbi.nlm.nih.gov/pubmed/28713755
http://dx.doi.org/10.4103/ams.ams_123_16
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author Chanukya, G. V.
Mengade, Manoj
Goud, Jagadishwar
Rao, I. Satish
Jain, Anuj
author_facet Chanukya, G. V.
Mengade, Manoj
Goud, Jagadishwar
Rao, I. Satish
Jain, Anuj
author_sort Chanukya, G. V.
collection PubMed
description Tumor-induced osteomalacia (TIO) is a subtype of paraneoplastic syndrome associated with hypophosphatemia due to renal phosphate wasting in adults. The humoral factor responsible for clinical picture known as fibroblast growth factor 23 (FGF23) is most often secreted by benign yet elusive mesenchymal tumors, difficult to localize, access, and excise completely; rarely, they are multiple and malignant. Paradoxical inappropriately normal or low levels of 1,25-dihydroxyvitamin D in the setting of hypophosphatemia is due to suppressive effect of FGF23. The following case report describes a 31-year-old male with symptoms of multiple fractures and severe muscle weakness, hypophosphatemia with elevated tubular maximum reabsorption of phosphate/glomerular filtration rate with low active Vitamin D, prompted assay for C-terminal FGF23, which was elevated multifold. The tumor was localized with whole body 68-Gadolinium DOTANOC positron emission tomography-computed tomography fusion scan in the left nasal cavity with ipsilateral maxillary antrum. It was excised through transnasal approach and found to be mesenchymal tumor on histopathology. At 1 week of follow-up, serum phosphate became normalized without supplementation. The patient is in follow-up for further measurement of FGF23 level and signs of recurrence. Because the occurrence of such a condition is rare and most often misdiagnosed or mismanaged for years, it is important to recognize this condition in differential diagnosis as potential curative surgical option is a reality.
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spelling pubmed-55025042017-07-14 Tumor-induced Osteomalacia: A Sherlock Holmes Approach to Diagnosis and Management Chanukya, G. V. Mengade, Manoj Goud, Jagadishwar Rao, I. Satish Jain, Anuj Ann Maxillofac Surg Case Report - Cysts and Tumors Tumor-induced osteomalacia (TIO) is a subtype of paraneoplastic syndrome associated with hypophosphatemia due to renal phosphate wasting in adults. The humoral factor responsible for clinical picture known as fibroblast growth factor 23 (FGF23) is most often secreted by benign yet elusive mesenchymal tumors, difficult to localize, access, and excise completely; rarely, they are multiple and malignant. Paradoxical inappropriately normal or low levels of 1,25-dihydroxyvitamin D in the setting of hypophosphatemia is due to suppressive effect of FGF23. The following case report describes a 31-year-old male with symptoms of multiple fractures and severe muscle weakness, hypophosphatemia with elevated tubular maximum reabsorption of phosphate/glomerular filtration rate with low active Vitamin D, prompted assay for C-terminal FGF23, which was elevated multifold. The tumor was localized with whole body 68-Gadolinium DOTANOC positron emission tomography-computed tomography fusion scan in the left nasal cavity with ipsilateral maxillary antrum. It was excised through transnasal approach and found to be mesenchymal tumor on histopathology. At 1 week of follow-up, serum phosphate became normalized without supplementation. The patient is in follow-up for further measurement of FGF23 level and signs of recurrence. Because the occurrence of such a condition is rare and most often misdiagnosed or mismanaged for years, it is important to recognize this condition in differential diagnosis as potential curative surgical option is a reality. Medknow Publications & Media Pvt Ltd 2017 /pmc/articles/PMC5502504/ /pubmed/28713755 http://dx.doi.org/10.4103/ams.ams_123_16 Text en Copyright: © 2017 Annals of Maxillofacial Surgery http://creativecommons.org/licenses/by-nc-sa/3.0 This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.
spellingShingle Case Report - Cysts and Tumors
Chanukya, G. V.
Mengade, Manoj
Goud, Jagadishwar
Rao, I. Satish
Jain, Anuj
Tumor-induced Osteomalacia: A Sherlock Holmes Approach to Diagnosis and Management
title Tumor-induced Osteomalacia: A Sherlock Holmes Approach to Diagnosis and Management
title_full Tumor-induced Osteomalacia: A Sherlock Holmes Approach to Diagnosis and Management
title_fullStr Tumor-induced Osteomalacia: A Sherlock Holmes Approach to Diagnosis and Management
title_full_unstemmed Tumor-induced Osteomalacia: A Sherlock Holmes Approach to Diagnosis and Management
title_short Tumor-induced Osteomalacia: A Sherlock Holmes Approach to Diagnosis and Management
title_sort tumor-induced osteomalacia: a sherlock holmes approach to diagnosis and management
topic Case Report - Cysts and Tumors
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5502504/
https://www.ncbi.nlm.nih.gov/pubmed/28713755
http://dx.doi.org/10.4103/ams.ams_123_16
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